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. 2017 May 13;25(8):1889–1899. doi: 10.1016/j.ymthe.2017.04.021

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© 2017 The Authors

This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

PMC Copyright notice

Fingolimod and Teriflunomide Attenuate Brain Atrophy in CLN1 and CLN3 Disease Models

(A) Total brain weights of 6-month-old Ppt1^+/+, Ppt1^−/−, fingolimod-treated Ppt1^−/− (+ FTY720), and teriflunomide-treated Ppt1^−/− (+ Teriflunomide) mice and 17-month-old Cln3^+/+, Cln3^−/−, fingolimod-treated Cln3^−/− (+ FTY720), and teriflunomide-treated Cln3^−/− (+ Teriflunomide) mice. Brain atrophy was attenuated in both models by treatment with fingolimod as well as with teriflunomide for 150 days (n = 5 mice/group). One-way ANOVA and Tukey’s post hoc tests. *p < 0.05, ***p < 0.001. (B) Representative light microscopy of cresyl violet-stained coronal brain sections of 6-month-old Ppt1^+/+, Ppt1^−/−, fingolimod-treated Ppt1^−/− (+ FTY720), and teriflunomide-treated Ppt1^−/− (+ Teriflunomide) mice. Note the reduction of brain atrophy in treated Ppt1^−/− mice. Scale bar, 1 mm. (C) Representative microscopy of autofluorescence (Af) in coronal sections of somatosensory cortex from 6-month-old Ppt1^+/+, Ppt1^−/−, fingolimod-treated Ppt1^−/− (+ FTY720), and teriflunomide-treated Ppt1^−/− (+ Teriflunomide) mice. There was no obvious difference regarding autofluorescence in treated Ppt1^−/− mice. Scale bar, 50 μm.